不明原因左心室肥厚(LVH)患者Fabry病的诊断困难:新的GLA基因突变是致病突变还是多态性?

IF 0.5 4区 医学 Q4 GENETICS & HEREDITY Balkan Journal of Medical Genetics Pub Date : 2023-07-01 DOI:10.2478/bjmg-2023-0010
N Aladağ, H Ali Barman, A Şipal, T Akbulut, M Özdemir, S Ceylaner
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摘要

法布里病(FD)是一种罕见的x连锁溶酶体鞘糖脂储存疾病。心脏受累可能包括左心室肥厚(LVH)、心律失常、传导异常、心力衰竭和瓣膜异常。本研究的目的是对不明原因LVH患者的FD进行基因分析。120例30岁以上经超声心动图诊断为特发性LVH的患者纳入研究。重度高血压、中度主动脉瓣狭窄、已知FD和常染色体显性肥厚性心肌病家族史的患者被排除在研究之外。所有患者均采用Sanger序列分析研究GLA基因突变。在本研究纳入的120例患者中,女性69例(58%),男性51例(42%)。平均年龄60.3±15.7岁。3例男性患者检测到GLA基因突变。检测到的突变如下:NM_000169.2:IVS6-10G>A (c.1000- 10g >A), NM_000169.2:c。937G>T (p.D313Y) (p.Asp313Tyr)和NM_000169.2:c。941A>T (p.K314M) (p.Lys314Met)。早期诊断对FD至关重要,可采用酶替代治疗。超声心动图诊断为特发性LVH患者的遗传筛查对FD的早期诊断和治疗具有重要意义。30岁以上的特发性LVH患者应筛查FD。在遗传筛选中可以检测到各种新的多态性。鉴定新的多态性对于了解FD的真正突变具有重要意义。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Difficulties in Diagnosing Fabry Disease in Patients with Unexplained Left Ventricular Hypertrophy (LVH): Is the Novel GLA Gene Mutation a Pathogenic Mutation or Polymorphism?

Fabry disease (FD) is an X-linked, lysosomal glycosphingolipid storage disorder that occurs very rarely. Cardiac involvement may comprise of left ventricular hypertrophy (LVH), arrhythmias, conduction abnormalities, heart failure and valvular abnormalities. The goal of this study was to conduct gene analysis for FD in patients suffering from unexplained LVH. 120 patients over the age of 30 who were diagnosed by echocardiography with idiopathic LVH were included in the study. Patients with severe hypertension, intermediate valve disease such as moderate aortic stenosis, known FD, and a family history of autosomal dominant hypertrophic cardiomyopathy were excluded from the study. GLA gene mutations were studied by Sanger sequence analysis in all patients. Of the 120 total patients included in this study, 69 were female (58%) and 51 were male (42%). The mean age was 60.3 ± 15.7. GLA gene mutations were detected in three male patients. The detected mutations are as follows: NM_000169.2:IVS6-10G>A (c.1000-10G>A), NM_000169.2:c.937G>T (p.D313Y) (p.Asp313Tyr) and NM_000169.2:c.941A>T (p.K314M) (p.Lys314Met). Early diagnosis is of vital importance in FD, which can be treated with enzyme replacement. Genetic screening in patients diagnosed with idiopathic LVH by echocardiography is important in the early diagnosis and treatment of FD. Patients over 30 years of age with idiopathic LVH should be screened for FD. Various new polymorphisms can be detected in genetic screening. Identifying new polymorphisms is important for knowing the true mutations in FD.

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期刊介绍: Balkan Journal of Medical Genetics is a journal in the English language for publication of articles involving all branches of medical genetics: human cytogenetics, molecular genetics, clinical genetics, immunogenetics, oncogenetics, pharmacogenetics, population genetics, genetic screening and diagnosis of monogenic and polygenic diseases, prenatal and preimplantation genetic diagnosis, genetic counselling, advances in treatment and prevention.
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